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      Transcatheter closure for baffle leak after Takeuchi repair of anomalous left coronary artery from the pulmonary artery: a case report

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          Abstract

          Introduction

          Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare but serious congenital coronary abnormality, Takeuchi repair is an alternative treatment option for this anomaly in certain situations, it is reported that baffle leak is the most common complication after Takeuchi repair, and some of this complication require surgical reoperation.

          Case presentation

          In this case, a 43-year-old woman diagnosed with ALCAPA underwent Takeuchi procedure 6 months ago, 4 months after the procedure, the patient complained of chest pain on exertion and was confirmed to have baffle leak, and then we treated this complication successfully by percutaneous transcatheter closure.

          Discussion

          Baffle leak is the most common complication after Takeuchi procedure of ALCAPA, some of them require surgical reoperation. In this case, we introduce a new method, percutaneous transcatheter closure, to treat the baffle leak. To our knowledge, this is the first reported case of transcatheter closure for baffle leak after Takeuchi repair, which may be an alternative treatment option for the baffle leak after Takeuchi repair of this rare congenital coronary anomaly.

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          Most cited references11

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          ALCAPA syndrome: not just a pediatric disease.

          Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) syndrome is a rare congenital coronary artery anomaly. There are two types of ALCAPA syndrome: the infant type and the adult type, each of which has different manifestations and outcomes. Infants experience myocardial infarction and congestive heart failure, and approximately 90% die within the 1st year of life. Rarely, ALCAPA syndrome manifests in adults; it may be an important cause of sudden cardiac death. Historically, ALCAPA syndrome was diagnosed at conventional angiography. However, the development of electrogardiographically gated multidetector computed tomographic (CT) angiography and magnetic resonance (MR) imaging enables accurate noninvasive imaging. At MR imaging and multidetector CT angiography, findings include direct visualization of the left coronary artery arising from the main pulmonary artery. Reversed flow from the left coronary artery into the main pulmonary artery may be seen at steady-state free-precession cine and fast cine phase-contrast MR imaging. Because of its ability to assess myocardial viability, which can be used as a prognostic factor to direct the need for surgical repair, MR imaging plays an important role in patient treatment. Restoration of a dual-coronary-artery system is the ideal surgical treatment for ALCAPA syndrome. (c) RSNA, 2009.
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            New surgical method for repair of anomalous left coronary artery from pulmonary artery.

            A new surgical method for repair of anomalous origin of the left coronary artery from the pulmonary trunk is described. The principle of the proposed technique is to transmit the oxygenated blood to the anomalous left coronary artery through surgically created aortopulmonary window and the internal tunnel created in the main pulmonary trunk. A 2-year-old boy in whom this anomaly was associated with mitral regurgitation caused by papillary muscle dysfunction was successfully treated by this new surgical method, and the deformed mitral valve was concomitantly replaced by a Hancock porcine xenograft.
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              Late complications after Takeuchi repair of anomalous left coronary artery from the pulmonary artery: case series and review of literature.

              Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare congenital heart lesion that if left untreated, results in high mortality. Surgical treatment options include reimplantation of the anomalous coronary ostia, use of extracardiac arterial blood supply, or the Takeuchi procedure. The Takeuchi procedure involves creation of an aortopulmonary window and an intrapulmonary tunnel that baffles the aorta to the ostium of the anomalous left coronary artery. The incidence of late complications following the Takeuchi repair is unknown. The goal of our study was to determine the long-term outcome after palliation of ALCAPA using the Takeuchi Procedure. A total of 9 patients with a history of ALCAPA palliated with the Takeuchi procedure were identified from our surgical database. Chart review was performed. The mean age at time of Takeuchi procedure was 49.6 months (range 5 weeks-14.6 years). There was one late death, of unknown cause. Of the remaining 8 patients, the mean length of follow-up after surgery was 15.9 years (13.5-19.7 years). All 8 survivors had some degree of main pulmonary artery stenosis in the area of the intrapulmonary baffle, with moderate stenosis in 2 and severe stenosis in 1. Three late survivors (38 %) had a baffle leak. Two patients (25 %) had decreased left ventricular systolic function and 3 (38 %) had developed at least moderate mitral valve regurgitation. Three of the 8 late survivors (38 %) required a reoperation for repair of mitral valve regurgitation, baffle leak, and main pulmonary artery (MPA) stenosis. Review of literature demonstrated similar complication rates and need for reoperation following the Takeuchi procedure. Compared with patients after direct reimplantation for ALCAPA at our institution, there was no significant difference in late survival or freedom from reoperation. The Takeuchi procedure is a method to establish a two-coronary repair for ALCAPA. Late complications are common, necessitating lifelong care in a center experienced with caring for adults with congenital heart disease.
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                Author and article information

                Journal
                Eur Heart J Case Rep
                Eur Heart J Case Rep
                ehjcr
                European Heart Journal: Case Reports
                Oxford University Press
                2514-2119
                June 2018
                29 March 2018
                29 March 2018
                : 2
                : 2
                : yty028
                Affiliations
                Center of Structural Heart Disease, Fuwai Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
                Author notes
                Corresponding author. Tel: 15600300802, Email: fwyyykai@ 123456sina.com or hhb1999@ 123456sina.com . This case report was reviewed by George Giannakoulas and Ina Michel-Behnke.
                Article
                yty028
                10.1093/ehjcr/yty028
                6177070
                9fece0a4-fbf5-46fd-aa3e-74b13ae2cb13
                © The Author(s) 2018. Published by Oxford University Press on behalf of the European Society of Cardiology.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

                History
                : 17 November 2017
                : 11 February 2018
                Page count
                Pages: 4
                Categories
                Case Reports

                case report,coronary anomaly,alcapa,takeuchi,baffle leak,transcatheter closure

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